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Tenth Case of Bilateral Hemifacial Spasm Treated by Microvascular

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Tenth Case of Bilateral Hemifacial Spasm Treated by Microvascular OPEN ACCESS Editor: Kazuhiro Hongo, M.D., SNI: Neurovascular For entire Editorial Board visit : Shinsui University, Matsomoto, http://www.surgicalneurologyint.com Japan Review Article Tenth case of bilateral hemifacial spasm treated by microvascular decompression: Review of the pathophysiology Warley Carvalho da Silva Martins1, Lucas Alverne Freitas de Albuquerque1, Gervásio Teles Cardoso de Carvalho1,2, Jules Carlos Dourado1, Marcos Dellaretti1,2,3, Atos Alves de Sousa1 1Department of Neurosurgery, Hospital Santa Casa de Belo Horizonte, 2Faculty of Medical Sciences of Minas Gerais, 3Department of Neurosurgery, Hospital das Clínicas de Belo Horizonte, Belo Horizonte, Minas Gerais, Brazil E‑mail: *Warley Carvalho da Silva Martins ‑ [email protected]; Lucas Alverne Freitas de Albuquerque ‑ [email protected]; Gervásio Teles Cardoso de Carvalho ‑ [email protected]; Jules Carlos Dourado ‑ [email protected]; Marcos Dellaretti ‑ [email protected]; Atos Alves de Sousa ‑ [email protected] *Corresponding author Received: 04 March 17 Accepted: 20 April 17 Published: 26 September 17 Abstract Background: Bilateral hemifacial spasm (BHFS) is a rare neurological syndrome whose diagnosis depends on excluding other facial dyskinesias. We present a case of BHFS along with a literature review. Methods: A 64‑year‑old white, hypertense male reported involuntary left hemiface contractions in 2001 (aged 50). In 2007, right hemifacial symptoms appeared, without spasm remission during sleep. Botulinum toxin type A application produced partial temporary improvement. Left microvascular decompression (MVD) was performed in August 2013, followed by right MVD in May 2014, with excellent results. Follow‑up in March 2016 showed complete cessation of spasms without medication. Results: The literature confirms nine BHFS cases bilaterally treated by MVD, a definitive surgical option with minimal complications. Regarding HFS pathophysiology, ectopic firing and ephaptic transmissions originate in the root Access this article online exit zone (REZ) of the facial nerve, due to neurovascular compression (NVC), Website: orthodromically stimulate facial muscles and antidromically stimulate the facial nerve www.surgicalneurologyint.com DOI: nucleus; this hyperexcitation continuously stimulates the facial muscles. These 10.4103/sni.sni_95_17 activated muscles can trigger somatosensory afferent skin nerve impulses and Quick Response Code: neuromuscular spindles from the trigeminal nerve, which, after transiting the Gasser ganglion and trigeminal nucleus, reach the somatosensory medial posterior ventral nucleus of the contralateral thalamus as well as the somatosensory cortical area of the face. Once activated, this area can stimulate the motor and supplementary motor areas (extrapyramidal and basal ganglia system), activating the motoneurons of the facial nerve nucleus and peripherally stimulating the facial muscles. This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 3.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms. For reprints contact: [email protected] How to cite this article: da Silva Martins WC, de Albuquerque LA, de Carvalho GT, Dourado JC, Dellaretti M, de Sousa AA. Tenth case of bilateral hemifacial spasm treated by microvascular decompression: Review of the pathophysiology. Surg Neurol Int 2017;8:225. http://surgicalneurologyint.com/Tenth-case-of-bilateral-hemifacial-spasm-treated-by-microvascular-decompression:-Review-of-the-pathophysiology/ © 2017 Surgical Neurology International | Published by Wolters Kluwer - Medknow Surgical Neurology International 2017, 8:225 http://www.surgicalneurologyint.com/content/8/1/225 Conclusions: We believe that bilateral MVD is the best approach in cases of BHFS. Key Words: Bilateral hemifacial spasm, botulinum toxin, microvascular decompression INTRODUCTION remained. In May 2014, he was submitted to right MVD, again presenting moderate postoperative peripheral facial Hemifacial spasm (HFS) is a common, involuntary, paresis, with full recovery in 3 weeks. All right HFS intermittent movement disorder induced in most ceased, but the previous right hypoacusis remained. cases by neurovascular compression (NVC) in the root He remained free from facial spasms until August 2014, exit zone (REZ) of the ipsilateral facial nerve in the when he began to experience mild relapse in the right brainstem.[5,19,92,94] Bilateral involvement of the face is a hemiface, with two to three fleeting spasms of the very rare event, ranging from 0.6 to 5% of cases of facial orbicularis muscle per hour and no episodes during sleep, spasm.[17,19,57,81,93,94] which had previously been an important complaint. His To our knowledge, this is the tenth case of bilateral HFS health status remained the same during 19 months of (BHFS) treated by microvascular decompression (MVD) follow‑up. In March 2016, he returned for evaluation to be described. A comprehensive review of the literature showing cessation of spasms without medication. The follows the case report. hearing deficits did not improve. MATERIALS AND METHODS/CASE RESULTS AND DISCUSSION MATERIAL Epidemiology Case report The annual incidence of HFS is 0.81 per 100,000 in A 64‑year‑old, white, hypertense male reported the onset women and 0.74 per 100,000 in men. The average of involuntary contractions of the left orbicularis muscle prevalence is 11 per 100,000 in the general population. in 2001 (aged 50), which progressed to contractions Women develop the disease in 14.5 per 100,000 of all the muscles in this hemiface. In 2007, symptoms individuals and men in 7.4 per 100,000 individuals, thus also appeared in the right orbicularis muscle and again female:male distribution is 2:1.[3,4,93] Prevalence increases progressed to all muscles in this hemiface. The patient with age, affecting 37.9 per 100,000 inhabitants in those reported no remission of spasms in either hemiface during over 70 years old.[73] sleep. Carbamazepine was prescribed (200 mg BID) with A systematic review of 5,685 cases of HFS submitted slight improvement in the symptoms initially, followed to MVD showed 69% were female and 31% were male. by relapses. Next, he was submitted to applications of Patient mean age at surgery was 54 (45–58) years old. The botulinum toxin type A (BTX‑A), which produced partial mean duration of symptoms was 6.5 (4.2–10.7) years.[63] temporary improvement. Among 6,029 patients with HFS submitted to MVD, The course of the disease included bilateral progressive 66.82% were female and 33.18% were male [Table 1]. hearing loss. In September 2012, he was submitted Comparisons between patients submitted to MVD and to audiometry, which showed mild to moderate those treated with BTX‑A showed they were similar in age, sensorineural loss (descending audiometric curve) in but the latter had a lower mean disease duration (6.7 vs. both ears. Magnetic resonance imaging (MRI) was 4.3 years, respectively).[93] performed in October 2012 and showed that the left vertebral artery followed a tortuous and elongated path, Cases of bilateral hemifacial spasm in the compressing and dislocating ipsilateral cranial nerves VII literature and VIII, compatible with neurovascular conflict, and To our knowledge, only 102 cases of BHFS have been discrete tortuosity of the right posterior inferior cerebellar reported [Table 2],[6,13,19,31,47,55‑57,72,81,86,92,94,100,105] and of artery (PICA), touching the brainstem along the REZ of these, only 10 cases were submitted to bilateral MVD, the VII‑VIII nerve complex on the right side. including our case.[6,13,81] The patient was submitted to left MVD in August 2013 In cases of BHFS, following a highly variable latency because the symptoms were more pronounced in this period of approximately 33–100 months, the spasms, hemiface. He evolved with complete cessation of spasms which initially begin in one hemiface, progress to the in the left hemiface, presenting moderate postoperative contralateral side.[55,94] In our case, the interval was peripheral facial paresis with progressive and full 6 years (72 months). The patient then goes on to present improvement in December, though the left hypoacusis involuntary painless asymmetric asynchronous facial Surgical Neurology International 2017, 8:225 http://www.surgicalneurologyint.com/content/8/1/225 Table 1: Epidemiology of patients with HFS Authors and year (location) Number of Females/ Left/right Mean age/years Mean duration of symptoms HFS/MVD Male (%) sides (%) until treatment/years Huang et al., 1992 (Taipei, Taiwan) 310 46/54 54.2/45.8 53.4 ‑ Barker et al., 1995 (Pittsburgh, USA) 648 65/35 60/40 52 8 Zhang et al., 1995 (Guangzhou, China) 300 56.3/43.7 44.7/55.3 ‑ 6 Payner and Tew, 1996 (Cincinnati, USA) 34 55.9/44.1 52.9/47.1 52 6.8 Ishikawa et al., 2001 (Saitama, Japan) 175 62.9/37.1 57.7/42.5 53 6.8 Samii et al., 2002 (Hannover, Germany) 143 62.2/37.8 56.6/43.4 54.5 6 Li, 2005 (Taipei, Taiwan) 545 ‑ 59.8/40.2 54.8 5.4 Yuan et al., 2005 (Beijing, China) 1,200 59.6/40.4 45.3/54.7 53 ? Han et al., 2009 (Sungnam, Korea) 1,642 77.3/22.7 ‑ 49.5 (3.4 <30 yrs) 7 Hyun et al., 2010 (Seoul, Korea) 1,174 71.1/28.9 50.2/49.8 48.8 5.6 Thirumala et al., 2011 (Pittsburgh, USA) 293 64.8/35.2 56/44 52.25 ‑ Rosenstengel et al., 2012 (Greifswald, 110 62.7/37.3 64.45/34.55 ‑ 8.1 Germany) Total or percentage (%) 6,574 66.82/33.18 52.43/47.57
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